tbuckley/cabot-search huggingface dataset:
Concurrent FDG-Avid and Non–FDG-Avid Pleomorphic Adenomas in the Submandibular Gland and Parapharyngeal Space
Authors: Soonchan Park, Young Chan Lee, Gou Young Kim, Chanwoo Kim
Journal: Clinical Nuclear Medicine (2023)

Abstract Pleomorphic adenoma is the most common benign tumor of salivary glands. Here we present an interesting case of concurrent pleomorphic adenomas in the parapharyngeal space and submandibular gland. The tumors showed stark differences in the imaging findings on FDG PET/CT and MRI. Pathology confirmed the diagnosis of pleomorphic adenomas with the different composition of the cellular component and chondromyxoid stroma. This case suggests that the difference in cellularity of pleomorphic adenomas can affect FDG uptake and diffusion-weighted MRI-derived apparent diffusion coefficient values.
FDA Approval Summary: Belzutifan for VHL Disease Tumors—Response
Authors: Jaleh Fallah, Chana Weinstock, Gautam U. Mehta, Michael H. Brave, William F. Pierce, Richard Pazdur, Abhilasha Nair, Daniel L. Suzman, Laleh Amiri-Kordestani
Journal: Clinical Cancer Research (2023)

Belzutifan received FDA approval for VHL-associated disease including central nervous system hemangioblastomas (CNS HB) in August 2021. The approval was based on results of the single-arm MK-6482 study that included 61 patients overall treated with belzutifan 120 mg orally daily, including 51 with CNS HB at baseline (per investigator assessment). The overall response rate for CNS HB in MK-6482-004 was 63% in 24 patients who had CNS HB with a measurable solid lesion with a median time to response of 3.1 months (range: 2.5–11; refs. 1, 2).Trinh and colleagues reported that a 54-year-old man with VHL disease developed bleeding in a previously undiagnosed hemangioblastoma in his spine 4 days after initiation of treatment with belzutifan (3). Intracranial hemorrhage is a known, but rare, serious complication of VHL disease–associated CNS HB (4). The package insert for belzutifan currently does not identify hemorrhage, including CNS hemorrhage, as an adverse reaction observed in patients receiving belzutifan in the setting of clinical trials.FDA review of the safety and efficacy of drugs does not end at marketing approval and the FDA continues to assess these in the postmarketing setting. The FDA Adverse Event Reporting System (FAERS) is a database that contains information on postmarketing adverse event, and medication error reports and product quality complaints resulting in adverse events. The database is designed to support the FDA's postmarketing safety surveillance program for drug and therapeutic biologic products (5). Health care professionals are encouraged to report the adverse events they observe with belzutifan and any other drug to the FDA via the FAERS database unless the adverse events are clearly due to disease progression or extraneous causes. FDA continually reassesses safety information and benefit-risk profile for approved drugs as more data are collected both from clinical trials and from the postmarketing setting to ensure safe and effective use. FDA encourages clinicians and patients to regularly refer to updated product labeling and Patient Package Insert for full information about safe and effective product use.See the original Letter to the Editor, p. 684No disclosures were reported.
Etiology of the Broad Avoidant Restrictive Food Intake Disorder Phenotype in Swedish Twins Aged 6 to 12 Years
Authors: Lisa Dinkler, Marie-Louis Wronski, Paul Lichtenstein, Sebastian Lundström, Henrik Larsson, Nadia Micali, Mark J. Taylor, Cynthia M. Bulik
Journal: JAMA Psychiatry (2023)

Avoidant restrictive food intake disorder (ARFID) is characterized by an extremely limited range and/or amount of food eaten, resulting in the persistent failure to meet nutritional and/or energy needs. Its etiology is poorly understood, and knowledge of genetic and environmental contributions to ARFID is needed to guide future research.To estimate the extent to which genetic and environmental factors contribute to the liability to the broad ARFID phenotype.This nationwide Swedish twin study includes 16 951 twin pairs born between 1992 and 2010 whose parents participated in the Child and Adolescent Twin Study in Sweden (CATSS) at twin age 9 or 12 years. CATSS was linked to the National Patient Register (NPR) and the Prescribed Drug Register (PDR). Data were collected from July 2004 to April 2020, and data were analyzed from October 2021 to October 2022.From CATSS, NPR, and PDR, all parent reports, diagnoses, procedures, and prescribed drugs that were relevant to the DSM-5 ARFID criteria were extracted when twin pairs were aged 6 to 12 years and integrated into a composite measure for the ARFID phenotype (ie, avoidant/restrictive eating with clinically significant impact, such as low weight or nutritional deficiency, and with fear of weight gain as an exclusion). In sensitivity analyses, autism and medical conditions that could account for the eating disturbance were controlled for. Univariate liability threshold models were fitted to estimate the relative contribution of genetic and environmental variation to the liability to the ARFID phenotype.Of 33 902 included children, 17 151 (50.6%) were male. A total of 682 children (2.0%) with the ARFID phenotype were identified. The heritability of ARFID was 0.79 (95% CI, 0.70-0.85), with significant contributions from nonshared environmental factors (0.21; 95% CI, 0.15-0.30). Heritability was very similar when excluding children with autism (0.77; 95% CI, 0.67-0.84) or medical illnesses that could account for the eating disturbance (0.79; 95% CI, 0.70-0.86).Prevalence and sex distribution of the broad ARFID phenotype were similar to previous studies, supporting the use of existing epidemiological data to identify children with ARFID. This study of the estimated genetic and environmental etiology of ARFID suggests that ARFID is highly heritable, encouraging future twin and molecular genetic studies.
The Humeral Metastasis as the Initial Presentation of Lung Adenocarcinoma Revealed by 18F-FDG PET/CT
Authors: Guozhu Hou, Pan Peng, Yiting Zhao, Rong Zheng
Journal: Clinical Nuclear Medicine (2022)

Lung adenocarcinoma with humeral metastasis as the initial presentation is rare. We reported FDG PET/CT findings of solitary humeral metastasis from adenocarcinoma of the lung in a 57-year-old man with the right upper arm pain as the initial presentation. MRI showed bone destruction of the right humerus with a soft tissue mass, suggesting the possibility of malignancy. FDG PET/CT showed that the right humeral mass had strong activity, and there were additional FDG lesions in the right armpit, left apical lung, and left adrenal gland, which was later confirmed as humeral metastasis from lung adenocarcinoma by pathological examination.
Vertebral Body Osteonecrosis Mimicking Malignant Disease on 18F-FDG PET
Authors: Kanishk Chaudhary, Stanley Ngai
Journal: Clinical Nuclear Medicine (2022)

We report a case of vertebral osteonecrosis after chemotherapy in a 24-year-old man with non-Hodgkin lymphoma, with transient avidity on 18F-FDG PET initially misinterpreted as recurrent extranodal disease. The patient demonstrated a partial metabolic response on midtreatment restaging PET; however, posttreatment PET showed an increase in uptake in T4 and T5 vertebrae, interpreted as recurrent vertebral disease. Repeat PET performed 9 days later showed resolution of thoracic vertebral uptake without interval treatment. On follow-up PET study, thoracic vertebrae demonstrated photopenia with sclerosis on CT, concerning for osteonecrosis. MRI features were concordant with this diagnosis.
The impact of primary ciliary dyskinesia on female and male fertility: a narrative review
Authors: Lydia Newman, Jagrati Chopra, Claire Dossett, Elizabeth Shepherd, Amelia Bercusson, Mary Carroll, Woolf Walker, Jane S Lucas, Ying Cheong
Journal: Human Reproduction Update (2023)

Abstract BACKGROUND Primary ciliary dyskinesia (PCD) is a genetic condition affecting the structure and function of sperm flagellum and motile cilia including those in the male and female reproductive tracts. Infertility is a commonly reported feature of PCD, but there is uncertainty as to how best to counsel patients on their fertility prognosis. OBJECTIVE AND RATIONALE This review aimed to summarize the prevalence of subfertility, possible underlying mechanisms, and the success of ART in men and women with PCD. The efficacy of ART in this patient group is relatively unknown and, hence, the management of infertility in PCD patients remains a challenge. There are no previous published or registered systematic reviews of fertility outcomes in PCD. SEARCH METHODS Systematic literature searches were performed in Medline, Embase, Cochrane Library, and PubMed electronic databases to identify publications between 1964 and 2022 reporting fertility outcomes in men and women with PCD. Publications were excluded if they reported only animal studies, where gender was not specified or where subjects had a medical co-morbidity also known to impact fertility. Quality of evidence was assessed by critical appraisal and application of an appraisal tool for cross-sectional studies. The primary outcomes were natural conception in men and women with PCD, and conception following ART in men and women with PCD. OUTCOMES A total of 1565 publications were identified, and 108 publications were included after screening by two independent researchers. The quality of available evidence was low. The exact prevalence of subfertility in PCD is unclear but appears to be higher in men (up to 83% affected) compared to women (up to 61% affected). Variation in the prevalence of subfertility was observed between geographic populations which may be explained by differences in underlying genotype and cilia function. Limited evidence suggests subfertility in affected individuals is likely caused by abnormal cilia motion in the fallopian tubes, endometrium and efferent ductules, and dysmotile sperm. Some men and women with PCD benefited from ART, which suggests its use should be considered in the management of subfertility in this patient group. Further epidemiological and controlled studies are needed to determine the predictors of fertility and optimal management in this patient group. WIDER IMPLICATIONS It is important that patients with PCD receive evidence-based counselling about the potential impact of their condition on their fertility prognosis and what management options may be available to them if affected. Understanding the pathophysiology and optimal management of subfertility in PCD will increase our understanding of the role of cilia and the impact of wider secondary ciliopathies on reproduction.
Cervical Lymph Node Metastasis From Recurrent Glioblastoma Detected by MRI and 2-[18F]FDG PET/CT
Authors: Marina Suárez-Piñera, Michal Pudis, Noelia Vilariño, Nuria Baixeras, Carlos Majós Torró
Journal: Clinical Nuclear Medicine (2023)

Abstract A 53-year-old man diagnosed with recurrent glioblastoma after multimodal treatment on a follow-up brain MRI. Because of a palpable mass in the neck, the patient underwent a whole-body 2-[ 18 F]FDG PET/CT, which revealed hypermetabolic laterocervical confluent lymphadenopathies. A nodal cervical biopsy indicated distant metastases from glioblastoma. Recent studies have confirmed dural meningeal lymphatics, as part of the glymphatic system, which provide clearance of interstitial solutes from the brain parenchyma into cervical lymphatics. Nodal cervical metastases from glioblastoma, using this pathway, are extremely rare and have been almost unthinkable some years ago.
High 18F-FDG Uptake in a Papillary Craniopharyngioma of the Third Ventricle
Authors: Hayato Kaida, Yuko Matsukubo, Sung-Woon Im, Nobuo Kashiwagi, Kazunari Ishii
Journal: Clinical Nuclear Medicine (2023)

Craniopharyngioma is a benign tumor classified as grade 1 by the World Health Organization Classification of Tumors of the Central Nervous System. We present a rare case of a high-18F-FDG-avidity papillary craniopharyngioma of the third ventricle. A 65-year-old man underwent CT and MRI examinations for gait disturbance, lower-limb weakness, and urinary incontinence, and an oval solid tumor that extended from the suprasellar region to the third ventricle was identified. 18F-FDG PET/CT showed high accumulation (SUVmax, 22.3) in the tumor. A transventricular endoscopic tumor biopsy led to the diagnosis of papillary craniopharyngioma.
The landscape of immune dysregulation in Crohn’s disease revealed through single-cell transcriptomic profiling in the ileum and colon
Authors: Lingjia Kong, Vladislav Pokatayev, Ariel Lefkovith, Grace T. Carter, Elizabeth A. Creasey, Chirag Krishna, Sathish Subramanian, Bharati Kochar, Orr Ashenberg, Helena Lau, Ashwin N. Ananthakrishnan, Daniel B. Graham, Jacques Deguine, Ramnik J. Xavier
Journal: Immunity (2023)
Application of radiomics to meningiomas: A systematic review
Authors: Ruchit V Patel, Shun Yao, Raymond Y Huang, Wenya Linda Bi
Journal: Neuro-Oncology (2023)

Abstract Background Quantitative imaging analysis through radiomics is a powerful technology to non-invasively assess molecular correlates and guide clinical decision-making. There has been growing interest in image-based phenotyping for meningiomas given the complexities in management. Methods We systematically reviewed meningioma radiomics analyses published in PubMed, Embase, and Web of Science until December 20, 2021. We compiled performance data and assessed publication quality using the radiomics quality score (RQS). Results A total of 170 publications were grouped into 5 categories of radiomics applications to meningiomas: Tumor detection and segmentation (21%), classification across neurologic diseases (54%), grading (14%), feature correlation (3%), and prognostication (8%). A majority focused on technical model development (73%) versus clinical applications (27%), with increasing adoption of deep learning. Studies utilized either private institutional (50%) or public (49%) datasets, with only 68% using a validation dataset. For detection and segmentation, radiomic models had a mean accuracy of 93.1 ± 8.1% and a dice coefficient of 88.8 ± 7.9%. Meningioma classification had a mean accuracy of 95.2 ± 4.0%. Tumor grading had a mean area-under-the-curve (AUC) of 0.85 ± 0.08. Correlation with meningioma biological features had a mean AUC of 0.89 ± 0.07. Prognostication of the clinical course had a mean AUC of 0.83 ± 0.08. While clinical studies had a higher mean RQS compared to technical studies, quality was low overall with a mean RQS of 6.7 ± 5.9 (possible range −8 to 36). Conclusions There has been global growth in meningioma radiomics, driven by data accessibility and novel computational methodology. Translatability toward complex tasks such as prognostication requires studies that improve quality, develop comprehensive patient datasets, and engage in prospective trials.
Erratum. 2. Classification and diagnosis of diabetes: <i>Standards of Care in Diabetes—2023</i>. Diabetes Care 2023;46(Suppl. 1):S19–S40
Authors: Nuha A. ElSayed, Grazia Aleppo, Vanita R. Aroda, Raveendhara R. Bannuru, Florence M. Brown, Dennis Bruemmer, Billy S. Collins, Marisa E. Hilliard, Diana Isaacs, Eric L. Johnson, Scott Kahan, Kamlesh Khunti, Jose Leon, Sarah K. Lyons, Mary Lou Perry, Priya Prahalad, Richard E. Pratley, Jane Jeffrie Seley, Robert C. Stanton, Robert A. Gabbay
Journal: Diabetes Care (2023)

In Table 2.3 of the article cited above, the hypertension risk factor was incorrectly defined as “≥140/90 mmHg or on therapy for hypertension”; this has been corrected to “≥130/80 or on therapy for hypertension.” More information on updated hypertension guideline recommendations can be found in Section 10 of Standards of Care in Diabetes—2023, “Cardiovascular Disease and Risk Management” (https://doi.org/10.2337/dc23-S010). The online version of the article (https://doi.org/10.2337/dc23-S002) has been updated to correct the error.
Recommendations for Epstein-Barr virus–based screening for nasopharyngeal cancer in high- and intermediate-risk regions
Authors: W K Jacky Lam, Ann D King, Jacob A Miller, Zhiwei Liu, Kelly J Yu, Melvin L K Chua, Brigette B Y Ma, Ming Yuan Chen, Benjamin A Pinsky, Pei-Jen Lou, John K S Woo, Wan-Lun Hsu, Julia Simon, Denise L Doolan, Tim Waterboer, Edwin P Hui, Hui Li, Raymond K Tsang, Kenneth C W Wong, Julian P Goh, Alexander C Vlantis, Qi Yong Ai, Lun M Wong, Victor Abdullah, Jin Ching Lin, Chien-Jen Chen, Ruth M Pfeiffer, Quynh-Thu Le, Anne W M Lee, Mingfang Ji, Sumei Cao, Jun Ma, Anthony T C Chan, K C Allen Chan, Allan Hildesheim
Journal: JNCI Journal of the National Cancer Institute (2023)

A meeting of experts was held in November 2021 to review and discuss available data on performance of Epstein-Barr virus (EBV)-based approaches to screen for early stage nasopharyngeal carcinoma (NPC) and methods for the investigation and management of screen-positive individuals. Serum EBV antibody and plasma EBV DNA testing methods were considered. Both approaches were found to have favorable performance characteristics and to be cost-effective in high-risk populations. In addition to endoscopy, use of magnetic resonance imaging (MRI) to investigate screen-positive individuals was found to increase the sensitivity of NPC detection with minimal impact on cost-effectiveness of the screening program.
Fungal feelings in the irritable bowel syndrome: the intestinal mycobiome and abdominal pain
Authors: Iam van Thiel, Wj de Jonge, Rm van den Wijngaard
Journal: Gut Microbes (2023)

Although the gut microbiota consists of bacteria, viruses, and fungi, most publications addressing the microbiota-gut-brain axis in irritable bowel syndrome (IBS) have a sole focus on bacteria. This may relate to the relatively low presence of fungi and viruses as compared to bacteria. Yet, in the field of inflammatory bowel disease research, the publication of several papers addressing the role of the intestinal mycobiome now suggested that these low numbers do not necessarily translate to irrelevance. In this review, we discuss the available clinical and preclinical IBS mycobiome data, and speculate how these recent findings may relate to earlier observations in IBS. By surveying literature from the broader mycobiome research field, we identified questions open to future IBS-oriented investigations.
PRP19 Enhances Esophageal Squamous Cell Carcinoma Progression by Reprogramming SREBF1-Dependent Fatty Acid Metabolism
Authors: Guang-Cong Zhang, Xiang-Nan Yu, Hong-Ying Guo, Jia-Lei Sun, Zhi-Yong Liu, Ji-Min Zhu, Tao-Tao Liu, Ling Dong, Xi-Zhong Shen, Jie Yin
Journal: Cancer Research (2023)

Lipid metabolism reprogramming is a recognized hallmark of cancer cells. Identification of the underlying regulators of metabolic reprogramming in esophageal squamous cell carcinoma (ESCC) could uncover potential therapeutic targets to improve treatment. Here, we demonstrated that pre-mRNA processing factor 19 (PRP19) mediates reprogramming of lipid metabolism in ESCC. Expression of PRP19 was significantly upregulated in multiple ESCC cohorts and was correlated with poor clinical prognosis. PRP19 promoted ESCC proliferation in vitro and in vivo. Upregulation of PRP19 enhanced fatty acid synthesis through sterol regulatory element-binding protein 1 (SREBF1), a major transcription factor of lipid synthase. Moreover, PRP19 enhanced the stability of SREBF1 mRNA in an N6-methyladenosine-dependent manner. Overall, this study shows that PRP19-mediated fatty acid metabolism is crucial for ESCC progression. Targeting PRP19 is a potential therapeutic approach to reverse metabolic reprogramming in patients with ESCC.Upregulation of pre-mRNA processing factor 19 (PRP19) contributes to esophageal squamous cell carcinoma progression by reprogramming SREBF1-dependent fatty acid metabolism, identifying PRP19 as a potential prognostic biomarker and therapeutic target.
Reports of Guillain-Barré Syndrome After COVID-19 Vaccination in the United States
Authors: Winston E. Abara, Julianne Gee, Paige Marquez, Jared Woo, Tanya R. Myers, Allison DeSantis, Jane A. G. Baumblatt, Emily Jane Woo, Deborah Thompson, Narayan Nair, John R. Su, Tom T. Shimabukuro, David K. Shay
Journal: JAMA Network Open (2023)

Because of historical associations between vaccines and Guillain-Barré syndrome (GBS), the condition was a prespecified adverse event of special interest for COVID-19 vaccine monitoring.To evaluate GBS reports to the Vaccine Adverse Event Reporting System (VAERS) and compare reporting patterns within 21 and 42 days after vaccination with Ad26.COV2.S (Janssen), BNT162b2 (Pfizer-BioNTech), and mRNA-1273 (Moderna) COVID-19 vaccines.This retrospective cohort study was conducted using US VAERS reports submitted during December 2020 to January 2022. GBS case reports verified as meeting the Brighton Collaboration case definition for GBS in US adults after COVID-19 vaccination were included.Receipt of the Ad26.COV2.S, BNT162b2, or mRNA-1273 COVID-19 vaccine.Descriptive analyses of GBS case were conducted. GBS reporting rates within 21 and 42 days after Ad26.COV2.S, BNT162b2, or mRNA-1273 vaccination based on doses administered were calculated. Reporting rate ratios (RRRs) after receipt of Ad26.COV2.S vs BNT162b2 or mRNA-1273 within 21- and 42-day postvaccination intervals were calculated. Observed-to-expected (OE) ratios were estimated using published GBS background rates.Among 487 651 785 COVID-19 vaccine doses, 17 944 515 doses (3.7%) were Ad26.COV2.S, 266 859 784 doses (54.7%) were BNT162b2, and 202 847 486 doses (41.6%) were mRNA-1273. Of 295 verified reports of individuals with GBS identified after COVID-19 vaccination (12 Asian [4.1%], 18 Black [6.1%], and 193 White [65.4%]; 17 Hispanic [5.8%]; 169 males [57.3%]; median [IQR] age, 59.0 [46.0-68.0] years), 275 reports (93.2%) documented hospitalization. There were 209 and 253 reports of GBS that occurred within 21 days and 42 days of vaccination, respectively. Within 21 days of vaccination, GBS reporting rates per 1 000 000 doses were 3.29 for Ad26.COV.2, 0.29 for BNT162b2, and 0.35 for mRNA-1273 administered; within 42 days of vaccination, they were 4.07 for Ad26.COV.2, 0.34 for BNT162b2, and 0.44 for mRNA-1273. GBS was more frequently reported within 21 days after Ad26.COV2.S than after BNT162b2 (RRR = 11.40; 95% CI, 8.11-15.99) or mRNA-1273 (RRR = 9.26; 95% CI, 6.57-13.07) vaccination; similar findings were observed within 42 days after vaccination (BNT162b2: RRR = 12.06; 95% CI, 8.86-16.43; mRNA-1273: RRR = 9.27; 95% CI, 6.80-12.63). OE ratios were 3.79 (95% CI, 2.88-4.88) for 21-day and 2.34 (95% CI, 1.83-2.94) for 42-day intervals after Ad26.COV2.S vaccination and less than 1 (not significant) after BNT162b2 and mRNA-1273 vaccination within both postvaccination periods.This study found disproportionate reporting and imbalances after Ad26.COV2.S vaccination, suggesting that Ad26.COV2.S vaccination was associated with increased risk for GBS. No associations between mRNA COVID-19 vaccines and risk of GBS were observed.
Epidemiology, clinical presentation, and outcomes of 620 patients with eosinophilia in the intensive care unit
Authors: Antoine Gaillet, Pierre Bay, Edwige Péju, Hafid Ait-Oufella, Elie Azoulay, Nacime Benchabane, Charles Cerf, Yves Cohen, Nicolas de Prost, Stanislas Faguer, Guillaume Geri, Steven Grangé, Jean-Emmanuel Kahn, Louis Kreitmann, Romaric Larcher, Guillaume Lefèvre, Asma Mabrouki, Armand Mekonsto-Dessap, Kewin Panel, Frédéric Pène, Marc Pineton de Chambrun, Jean-Pierre Quenot, Yacine Tandjaoui-Lambiotte, Jean-Francois Timsit, Antoine Vieillard-Baron, Auguste Dargent, Antoine Herault, Matthieu Groh
Journal: Intensive Care Medicine (2023)